Expansion of the ophthalmic phenotype of SPINT2-related syndromic congenital sodium diarrhea

Am J Med Genet A. 2021 Apr;185(4):1270-1274. doi: 10.1002/ajmg.a.62094. Epub 2021 Feb 5.

Abstract

A 5-year-old girl presented with treatment-refractory dry eye and recurrent episodes of eye pain. She had been previously diagnosed with syndromic congenital sodium diarrhea (SCSD) caused by a pathogenic variant in SPINT2. Her local pediatric ophthalmologist had made the diagnosis of severe dry eye with corneal erosions, based on which, we arranged an eye exam under anesthesia (EUA) and punctal plug placement. Anterior segment optical coherence tomography (OCT) and corneal photographs were taken during the procedure. There are reports describing similar ophthalmic findings in this syndrome. However, to the best of our knowledge, this is the first case report to document OCT imaging and corneal photographs in a patient with SCSD, which we feel expands the ophthalmic phenotype of this rare genetic disorder.

Keywords: SPINT2; optical coherence tomography; photophobia; recurrent corneal erosions; syndromic congenital sodium diarrhea.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / diagnosis
  • Abnormalities, Multiple / diagnostic imaging
  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / pathology
  • Child, Preschool
  • Cornea / metabolism
  • Cornea / pathology
  • Diarrhea / congenital*
  • Diarrhea / diagnosis
  • Diarrhea / diagnostic imaging
  • Diarrhea / genetics
  • Diarrhea / pathology
  • Humans
  • Membrane Glycoproteins / genetics*
  • Metabolism, Inborn Errors / diagnosis
  • Metabolism, Inborn Errors / diagnostic imaging
  • Metabolism, Inborn Errors / genetics*
  • Metabolism, Inborn Errors / pathology
  • Mutation / genetics
  • Phenotype
  • Sodium / metabolism*
  • Tomography, Optical Coherence / methods

Substances

  • Membrane Glycoproteins
  • SPINT2 protein, human
  • Sodium

Supplementary concepts

  • Diarrhea 3, Secretory Sodium, Congenital